Generalized Verrucosis Revealing a Life-Threatening and Unlabeled T-Cell Lymphopenia Associated With Autoimmune Hemolytic Anemia: A Case Report and Review of Literature - Université Grenoble Alpes Accéder directement au contenu
Article Dans Une Revue Journal of Pediatric Hematology/Oncology Année : 2020

Generalized Verrucosis Revealing a Life-Threatening and Unlabeled T-Cell Lymphopenia Associated With Autoimmune Hemolytic Anemia: A Case Report and Review of Literature

Résumé

Primary immunodeficiencies are inherited disorders, which may be revealed in the context of autoimmune hemolytic anemia (AIHA). We report the case of a girl presenting with an enterovirus-related AIHA. Despite being in complete remission for her anemia after treatment, the initial CD4/CD8 lymphopenia dramatically worsened with time. Its sole clinical presentation was generalized verrucosis. Cellular quantitative and functional immunodeficiency was evidenced but no known molecular defect was identified despite extensive workup. This unlabeled profound naive T-lymphopenia was cured by bone marrow transplantation. No similar case was ever described in the scientific literature. Patients with AIHA and/or generalized verrucosis should be screened for primary immunodeficiency, before initiating any immunomodulatory treatment.
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hal-04556617 , version 1 (23-04-2024)

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Magali Marxgut, Dalila Adjaoud, Nathalie Aladjidi, Corinne Armari, Léa Marxgut, et al.. Generalized Verrucosis Revealing a Life-Threatening and Unlabeled T-Cell Lymphopenia Associated With Autoimmune Hemolytic Anemia: A Case Report and Review of Literature. Journal of Pediatric Hematology/Oncology, 2020, 42 (8), pp.e745-e749. ⟨10.1097/MPH.0000000000001599⟩. ⟨hal-04556617⟩

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